At present, the rate of extrapulmonary tuberculosis is gradually increasing among all tuberculosis cases. The secondary tuberculosis of oral cavity represents 0.2-1.5% of all cases with extrapulmonary tuberculosis. Although incidence of oral lesions secondary to pulmonary tuberculosis ranges from 0.4% to 1.5%, primary tuberculosis of the tongue occurs very rare, and it is published only as isolated case reports. A male patient presented to our dermatology clinic with complaints of painful ulcerated white plaques on the tip of his tongue lasting for three months, and an excisional biopsy showed granulomatous inflammation with caseification necrosis. Pulmonary and systemic examination showed no evidence of pulmonary or other extra-pulmonary tuberculosis, and he was initiated therapy for diagnosis of primary tuberculosis of the tongue. We found that the lesion completely resolved during the follow up visit after 1 month. Primary tuberculosis of the tongue should be for differential diagnosis of resistant painful oral lesions.